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A Novel Missense Mutation Affecting the N-terminal Domain of SAP Protein in X-linked Lymphoproliferative Disease

We have revealed a novel SH2D1A gene mutation in a patient with XLP resulting in fulminant refractory EBV-driven HLH, which is a recognized severe complication

Citation:
Furlong E, Carter TL, Williams A, Wong M, Ramachandran S, Kotecha RS. A Novel Missense Mutation Affecting the N-terminal Domain of SAP Protein in X-linked Lymphoproliferative Disease. Journal of Pediatric Hematology/Oncology. 2019;41(8):e550-e1

Abstract:
X-linked lymphoproliferative disease (XLP) is a rare primary immunodeficiency characterized by the inability of the affected individual to clear Epstein-Barr virus (EBV) resulting in hemophagocytic lymphohistiocytosis (HLH) in 55% to 76% of male individuals carrying mutations in SH2D1A (XLP-1) or XIAP (XLP-2). Here, we reveal a novel SH2D1A mutation in a child with XLP.